Wheat (Triticum aestivum L.) deletion (del) stocks are valuable tools for the physical mapping of molecular markers and genes to chromosome bins delineated by 2 adjacent deletion breakpoints. The wheat deletion stocks were produced by using gametocidal genes derived from related Aegilops species. Here, we report on the origin, structure, and behavior of a highly rearranged chromosome 1BS-4. The cytogenetic and molecular marker analyses suggest that 1BS-4 resulted from 2 breakpoints in the 1BS arm and 1 breakpoint in the 1BL arm. The distal segment from 1BS, except for a small deleted part, is translocated to the long arm. Cytologically, chromosome 1BS-4 is highly stable, but shows a unique meiotic pairing behavior. The short arm of 1BS-4 fails to pair with a normal 1BS arm because of lack of homology at the distal ends. The long arm of 1BS-4 only pairs with a normal 1BS arm within the distal region translocated from 1BS. Therefore, using the 1BS-4 deletion stock for physical mapping will result in the false allocation of molecular markers and genes proximal to the breakpoint of 1BS-4.